This project expands the PI's research on fragile X syndrome (FXS), which has focused on the development of language and other aspects of behavior in adolescents with FXS, to include the investigation of emotional well-being in mothers of these adolescents. Preliminary studies suggest that these mothers are at risk for lower levels of emotional well-being and that their well-being is related to rates of maladaptive behavior displayed by the son or daughter with FXS. These preliminary studies, however, are limited by a lack of longitudinal data necessary to establish cause-and-effect relationships, by small sample sizes that precluded inclusion of variables reflecting important characteristics of the child, the mother, and the contexts of development, and by a lack of adequate genetic characterization of the mothers. The proposed project will build on these findings and make use of the existing cohort to facilitate recruitment. Mothers and adolescents will be assessed in a short-term longitudinal study, with 2 data collection points separated by 1 year. Self-report informant report, and direction observation will be used. Mother genetic status will be determined as well. We will determine the extent to which (1) the maladaptive behaviors of the adolescent with FXS compromise maternal emotional well-being; (2) maternal emotional well-being affects the development of maladaptive behavior in the adolescent with FXS; and (3) whether the relations between maternal well-being and adolescent maladaptive behavior are moderated by maternal genetic status, adolescent gender, and family context. This project will pave the way for future, larger studies, involving representative samples of families with a more comprehensive assessment of the impacts of FXS on mothers, adolescents, and other family members and the processes by which those effects occur.

Agency
National Institute of Health (NIH)
Institute
Eunice Kennedy Shriver National Institute of Child Health & Human Development (NICHD)
Type
Small Research Grants (R03)
Project #
1R03HD048884-01A1
Application #
6967045
Study Section
Pediatrics Subcommittee (CHHD)
Program Officer
Vitkovic, Ljubisa
Project Start
2005-07-01
Project End
2007-06-30
Budget Start
2005-07-01
Budget End
2006-06-30
Support Year
1
Fiscal Year
2005
Total Cost
$71,532
Indirect Cost
Name
University of Wisconsin Madison
Department
Pediatrics
Type
Other Domestic Higher Education
DUNS #
161202122
City
Madison
State
WI
Country
United States
Zip Code
53715
Hauser, Claire T; Kover, Sara T; Abbeduto, Leonard (2014) Maternal well-being and child behavior in families with fragile X syndrome. Res Dev Disabil 35:2477-86
Hartley, Sigan L; Seltzer, Marsha Mailick; Head, Lara et al. (2012) Psychological Well-being in Fathers of Adolescents and Young Adults with Down Syndrome, Fragile X Syndrome, and Autism. Fam Relat 61:327-342
Seltzer, Marsha Mailick; Abbeduto, Leonard; Greenberg, Jan S et al. (2009) Biomarkers in the Study of Families of Individuals with Developmental Disabilities. Int Rev Res Ment Retard 37:213-249
Head, Lara S; Abbeduto, Leonard (2007) Recognizing the role of parents in developmental outcomes: a systems approach to evaluating the child with developmental disabilities. Ment Retard Dev Disabil Res Rev 13:293-301